M J L Mastboom1, E L Staals2, F G M Verspoor3, A J Rueten-Budde4, S Stacchiotti5, E Palmerini2, G R Schaap6, P C Jutte7, W Aston8, A Leithner9, D Dammerer10, A Takeuchi11, Q Thio12, X Niu13, J S Wunder14, M A J van de Sande1. 1. Orthopaedic Surgery, Leiden University Medical Center, Leiden, the Netherlands. 2. Orthopaedic Surgery (E.L.S.) and Medical Oncology (E.P.), Musculoskeletal Oncology Department, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy. 3. Orthopaedic Surgery, Radboud University Nijmegen Medical Center, Nijmegen, the Netherlands. 4. Mathematical Institute, Leiden University, Leiden, the Netherlands. 5. Medical Oncology, Fondazione IRCCS Istituto Nazionale dei Tumori, Milano, Italy. 6. Orthopaedic Surgery, Academic Medical Center, Amsterdam, the Netherlands. 7. Department of Orthopaedics, University Medical Center, University of Groningen, Groningen, the Netherlands. 8. Orthopaedic Surgery, Royal National Orthopaedic Hospital, London, United Kingdom. 9. Department of Orthopaedic Surgery, Medical University Graz, Graz, Austria. 10. Orthopaedic Surgery, Medical University of Innsbruck, Innsbruck, Austria. 11. Orthopaedic Surgery, Kanazawa University Graduate School of Medical Sciences, Kanazawa, Japan. 12. Orthopaedic Surgery, Massachusetts General Hospital Harvard, Boston, Massachusetts. 13. Department of Orthopaedic Oncology, Beijing Jishuitan Hospital, Beijing, China. 14. University Musculoskeletal Oncology Unit, Mount Sinai Hospital, Toronto, Ontario, Canada.
Abstract
BACKGROUND: Localized-type tenosynovial giant cell tumor (TGCT) is a rare, neoplastic disease with only limited data supporting treatment protocols. We describe treatment protocols and evaluate their oncological outcome, complications, and functional results in a large multicenter cohort of patients. A secondary study aim was to identify factors associated with local recurrence after surgical treatment. METHODS: Patients with histologically proven localized TGCT of a large joint were included if they had been treated between 1990 and 2017 in 1 of 31 tertiary sarcoma centers. Of 941 patients with localized TGCT, 62% were female. The median age at initial treatment was 39 years, and the median duration of follow-up was 34 months. Sixty-seven percent of the tumors affected the knee, and the primary treatment at the tertiary center was 1-stage open resection in 73% of the patients. Proposed factors for predicting a first local recurrence after treatment in the tertiary center were tested in a univariate analysis, and those that demonstrated significance were subsequently included in a multivariate analysis. RESULTS: The localized TGCT recurred in 12% of all cases, with local-recurrence-free rates at 3, 5, and 10 years of 88%, 83%, and 79%, respectively. The strongest factor for predicting recurrent disease was a prior recurrence (p < 0.001). Surgical treatment decreased pain and swelling in 71% and 85% of the patients, respectively, and such treatment was associated with complications in 4% of the patients. Univariate and multivariate analyses of the patients who had not undergone therapy previously yielded positive associations between local recurrence and a tumor size of ≥5 cm versus <5 cm (hazard ratio [HR] = 2.50; 95% confidence interval [CI] = 1.32 to 4.74; p = 0.005). Arthroscopy (versus open surgery) was significantly associated with tumor recurrence in the univariate analysis (p = 0.04) but not in the multivariate analysis (p = 0.056). CONCLUSIONS: Factors associated with recurrence after resection of localized-type TGCT were larger tumor size and initial treatment with arthroscopy. Relatively low complication rates and good functional outcomes warrant an open approach with complete resection when possible to reduce recurrence rates in high-risk patients. LEVEL OF EVIDENCE: Therapeutic Level III. See Instructions for Authors for a complete description of levels of evidence.
BACKGROUND: Localized-type tenosynovial giant cell tumor (TGCT) is a rare, neoplastic disease with only limited data supporting treatment protocols. We describe treatment protocols and evaluate their oncological outcome, complications, and functional results in a large multicenter cohort of patients. A secondary study aim was to identify factors associated with local recurrence after surgical treatment. METHODS:Patients with histologically proven localized TGCT of a large joint were included if they had been treated between 1990 and 2017 in 1 of 31 tertiary sarcoma centers. Of 941 patients with localized TGCT, 62% were female. The median age at initial treatment was 39 years, and the median duration of follow-up was 34 months. Sixty-seven percent of the tumors affected the knee, and the primary treatment at the tertiary center was 1-stage open resection in 73% of the patients. Proposed factors for predicting a first local recurrence after treatment in the tertiary center were tested in a univariate analysis, and those that demonstrated significance were subsequently included in a multivariate analysis. RESULTS: The localized TGCT recurred in 12% of all cases, with local-recurrence-free rates at 3, 5, and 10 years of 88%, 83%, and 79%, respectively. The strongest factor for predicting recurrent disease was a prior recurrence (p < 0.001). Surgical treatment decreased pain and swelling in 71% and 85% of the patients, respectively, and such treatment was associated with complications in 4% of the patients. Univariate and multivariate analyses of the patients who had not undergone therapy previously yielded positive associations between local recurrence and a tumor size of ≥5 cm versus <5 cm (hazard ratio [HR] = 2.50; 95% confidence interval [CI] = 1.32 to 4.74; p = 0.005). Arthroscopy (versus open surgery) was significantly associated with tumor recurrence in the univariate analysis (p = 0.04) but not in the multivariate analysis (p = 0.056). CONCLUSIONS: Factors associated with recurrence after resection of localized-type TGCT were larger tumor size and initial treatment with arthroscopy. Relatively low complication rates and good functional outcomes warrant an open approach with complete resection when possible to reduce recurrence rates in high-risk patients. LEVEL OF EVIDENCE: Therapeutic Level III. See Instructions for Authors for a complete description of levels of evidence.
Authors: J Lopez-Bastida; I Aranda-Reneo; B Rodríguez-Sánchez; L M Peña-Longobardo; X Ye; P Laeis; E M Fronk; E Palmerini; A Leithner; M A J Van de Sande Journal: Orphanet J Rare Dis Date: 2021-07-02 Impact factor: 4.123